Abstract
Purpose:
To evaluate the clinical outcome and safety of treatment with Adalimumab in patients with Vogt-Koyanagi-Harada (VKH) disease with refractory uveitis.
Methods:
Patients with VKH disease treated with Adalimumab for at least 6 months, in whom conventional therapy had failed were identified from April 2006 to April 2013. Patients in chronic stage treated with corticosteroids and immunosuppressives with active anterior and / or posterior inflammation were included. Adalimumab was administered on a compassionate off-label use basis at the dose of 40 mg subcutaneously every other week . The main outcome measures were BCVA, anterior segment inflammation (SUN) , efficacy of treatment in anterior and posterior inflammation, steroid sparing effect and reduction in concomitant immunosuppressive therapy.
Results:
Fourteen patients , 4 male and 10 female , with a mean age of 23.07 ± 8.51 years ( r= 11-39 ) with a mean follow-up of 42.35 ± 37.55 months (r=8-132)and a mean duration of adalimumab treatment (AT) of 14.57 ± 11.54 months ( r=6-45) were included in this study. Regarding the systemic corticosteroid, the treatment with AT at 6 months was statistically significantly lower than the dose at baseline (Wilcoxon signed rank test, P= 0.0037). Furthermore, the dose at 12 months was statistically significantly lower than the dose at 6 months of AT( Wilcoxon signed rank test , P= 0.0157). Out of 14 patients,11 (78.57%) had IMT at baseline whereas 5 out of 14 (35.71%) had IMT at 6 months of AT (Mc Nemar's Chi square test = 4.50 ; P= 0.0339) , showing a trend toward statistical significance (p= 0.07). The comparison between anterior inflammation in OD previous to AT and the one observed at 6 months revealed a statistically significant difference with the binomial test for one sided (P=0.0001) and for two sided (P=0.0002) . The comparison between the anterior inflammation in OS previous to AT and the one observed at 6 months revealed a statistically significant difference with the binomial test for one sided(P=0.0002) and for two sided (P=0.0005). Four patients had recurrences within the first three months after the withdrawal of medication.
Conclusions:
Our study showed the significant response to Adalimumab in 14 patients with diagnosis of VKH disease who were refractory to standard IMT. This anti-TNF agent seemed to be an effective and well-tolerated treatment for these patients.
Keywords: 557 inflammation •
746 uveitis-clinical/animal model •
432 autoimmune disease